Case Report Knee Arthrodesis with Autologous Patella Graft for Treatment of Charcot Knee with Severe Bone Loss: a Case Report

Int J Clin Exp Med 2019;12(1):1038-1045 www.ijcem.com /ISSN:1940-5901/IJCEM0079332

Case Report Knee arthrodesis with autologous patella graft for treatment of Charcot knee with severe bone loss: a case report

Zhi Chen, Yijie Shao, Chao Gao, Huilin Yang, Ming Xu

Department of Orthopaedic Surgery, The First Affiliated Hospital of Soochow University, No. 188 Shizi Street, Suzhou 215000, Jiangsu Province, P. R. China Received May 9, 2018; Accepted December 11, 2018; Epub January 15, 2019; Published January 30, 2019

Abstract: Background: Charcot arthropathy refers to a progressive destructive joint disease associated with nerve damage and periarticular insensitivity. Diabetes, tabes dorsalis, syringomyelia are the main causes. Case report: Herein we present the case of a 60 year old male with Charcot knee complicated by tabes dorsalis, in which posterior tibial bone loss, joint instability, bone fragility and susceptibility to infection were intractable problems. Knee arthrodesis with double locking compression plates were performed using autologous patella graft to fill in the bone defect and structurally reconstruct the tibial plateau, providing increased surface area for bony union. Findings obtained 10 months postoperatively showed maintenance of solid fusion with no complications. Conclusion: The technique described here appears to be safe and reproducible based on the current results, providing significant functional improvements in the patient with Charcot arthropathy. This case indicates that the technique could be an option in the operative management of Charcot knee with severe bone loss.

Keywords: Charcot arthropathy, tabes dorsalis, knee arthrodesis, bone loss, autologous patella graft

Introduction Tabes dorsalis is a form of tertiary parenchymal neurosyphilis that arises in 2% to 9% of individ- Charcot arthropathy was first described 140 uals with untreated syphilis and has an incuba- years ago by J.M. Charcot but it was not until tion period of 3 to 50 years post-infection [3]. 1936 that W.R. Jordan established the associa- The lesion principally affects the posterior fu- tion between Charcot arthropathy and diabetes niculus of the spinal cord, with cranial nerves mellitus, which is the leading cause of the con- also vulnerable. Clinical manifestations of ta- dition worldwide today [1]. Although most com- bes dorsalis may take decades to appear after monly observed in the foot and ankle, there is a the initial infection. Following more organized predilection for other sites including the knee, public health measures and the introduction of hip, shoulder and spine, depending on the un- penicillin in the 1950 s, the incidence of neuro- derlying etiology [2]. There is a lack of specific- syphilis has declined significantly and as a ity in the manifestations of Charcot arthropathy result, tabes dorsalis has become a rare dis- that distinguish it from other similar diseases. ease [4]. The incidence of Charcot arthropathy Besides joint swelling and deformity, lack of in patients suffering from tabes dorsalis ranges pain and sensory loss can be prominent symp- from 6% to 10%, which is an unneglectable toms. The absence of appropriate nociception number [5]. leads to excessive overload bearing or repeti- tive articular cartilage damage and bone de- Generally, total knee arthroplasty (TKA) is not fects, eventually leading to fracture or joint dis- recommended as a therapy for Charcot arthrop- location. Therefore, Charcot arthropathy should athy due to the high risk of internal fixation fail- be taken into consideration as soon as possi- ure. Over the last few years, these traditional ble when eliminating the possibility of other views have been questioned, with an increasing diseases. number of reports of Charcot knee arthropathy Knee arthrodesis with patella graft treating Charcot arthropathy

Case presentation

A 60-year old male was pre- sented to our hospital with a swollen left knee and restrict- ed movement since an initial sprain he had experienced while walking down the street 7 days prior. He was unable to stand or walk, but did not feel any pain. His condition gradu- ally deteriorated until he was finally admitted to our hospital. In the last 4 years, he had consulted numerous clinics with a long history of back- aches and flash pain in his lower limbs. However, the treat- ments appeared ineffective and there had been no clear Figure 1. A. First dislocation occurred 3 days after operation, the artificial femoral head dislocated from the ball and socket joint; B. Second occur- diagnosis. The patient had re- rence of dislocation was 1 month after operation, the acetabulum prosthe- ceived a bipolar femoral head sis dislocated together with artificial femoral head from acetabular fossa; C. replacement (BFHR) in anoth- The third dislocation happened 6 months postoperatively, which was simi- er hospital 2 years previously lar with the second time; D. Anteroposterior radiograph of bilateral hip joint due to a right femoral neck after total hip replacement showed favorable position of prosthesis, while fracture. The affected side dis- scattered bony fragments were visible around the hip joint. located on 3 occasions (Figure 1A-C) following the operation Table 1. Significant laboratory reports due to an unknown cause, with correction Test result Reference value achieved after one manual traction reduction and one open reduction procedure successive- WBC 8390 3500-9500/μl ly. By the time the third dislocation happened, Hb 111 130-175 g/L total hip replacement (THR) was ultimately per- ESR 62 0-15 mm/h formed 6 months after BFHR in another medi- CRP 46.1 0-8 mg/L cal institution (Figure 1D). Unexpectedly, right TPPA ++ - hip luxation occurred again only 2 months after RPR ++ - THR and was cured by manual traction reduc- Note: WBC, White blood cell; Hb, Hemoglobin; ESR, tion. The prosthesis remained in position until Erythrocyte sedimentation rate; CRP: C-reactive protein; this present injury. The patient confirmed that TPPA, Treponema pallidum antibodies; RPR, Rapid he did not have a history of diabetes, blood plasma reagin. Serological tests showed significant posi- transfusions, or genetic diseases. tive results for RPR and TPPA besides mild anemia and inflammation. In terms of physical findings, the passive and active range of motion of the left knee was -20°/130° in flexion without tenderness at the being treated with TKA. However, the curative joint spaces. The manual varus and valgus effects of TKA requires further study [6]. Joint stress test demonstrated apparent instability arthrodesis remains the conventional choice of while the anterior and posterior drawer test surgical treatment in most instances [7]. The showed positive results. On neurological find- major challenges when planning surgery in- ings, a clear loss in superficial sensation and clude severe bone loss, quality of soft tissue, bathyesthesia below the level of Th10 were and lack of healthy bone. For knee arthrodesis, observed in addition to the loss of bilateral ten- although orthobiologics and fibula grafts have don reflexes of the lower limbs. Muscular been widely used as an onlaystrut graft, the strength of the bilateral limbs was grade V with use of the patella in the reconstruction of the normal muscular tension and no induction of tibial plateau has not been reported. pathological signs.

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Laboratory data indicated an appreciable rise in erythrocyte sedimentation rate (ESR) and level of C-reactive protein (CRP) besides mild anemia (Table 1). Both treponema pallidum antibody (TPPA) and rapid plasma regain (RPR) results were positive (++) on a serological test, providing significant evidence of syphilis infection.

Plain radiography revealed anterior dislocation of the left knee in addition to fracture of the left tibial plateau and ca- put fibulae Figure ( 2). Com- puted tomography (CT) demonstrated a large bone defect of the posterior tibial plateau Figure 2. Plain radiography demonstrates anterior dislocation of left knee, and massive bone fragments fracture of tibial plateau and capitula fibula. around the knee joint were also visible (Figure 3). It is difficult to precisely classify the fracture based on current the- ories. Magnetic resonance imaging (MRI) showed the pres- ence of a bucket-handle me- niscus tear, posterior cruciate ligament (PCL) injury with severe synovitis and arthroede- ma (Figure 4). An MRI of the whole spine displayed mild intervertebral disc bulge at C5-C7 and L1-L3, but no evidence of syringomyelia, men- ingomyelocele or related diseases (Figure 5). An emission computed tomography (ECT) scan performed at another hospital 2.5 years previously showed a concentration of ra- dionuclide and abnormal bone metabolism in the right hip and left knee, but the results did not receive much atten- tion at the time (Figure 6).

According to these findings, his condition was diagnosed as Charcot arthropathy cau- sed by tabes dorsalis, with the Figure 3. Preoperative computed tomography shows comminuted tibial plateau fracture, large bone defects of posterior tibial plateau and mass bone right hip and left knee being fragments around the joint. affected. The patient received

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ed to 240 mmHg, skin pre- pared with entoiodine and Figure 4. Magnetic reso- antibiotic prophylaxis was us- nance imaging of left knee ed intravenously. A mid-longi- displays injury of posterior cruciate ligament, bucket- tudinal incision bypassing the handle tear of medial me- patella was created extending niscus, accompanied with distally to the tibial tubercle synovial edema and hy- (Figure 7A). Dissection of the drarthrosis. knee joint cavity revealed fea- tures consistent with pathological fracture, including mass blood clots around the joint and multiple brittle bony fragments without vascularity. La- rge bone defects on the posterior aspect of the tibial plateau were observed (Figure 7B). To prepare the femur and tibia for arthrodesis, osteotomy wa- s performed so that the knee was aligned with 10° of flexion, 5° of valgus and 5° of ex- torsion angulation, in which position intraoperative X-ray imaging proved that lower limb alignment was satisfactory. An osteotomy mold such as is employed for TKA, was used to ensure the accuracy of osteotomy. The resected patella was selected as an autologous bone graft to rebuild the structure of the tibial plateau. After removing the cartilaginous surface of the patella, it was split into two equal parts along the mid-longitudinal line with an oscillat- ing saw followed by meticu- lous clipping in order to fit into the bone defect (Figure 7C). After adjusting to an appropri- Figure 5. Magnetic resonance imaging of whole spine reveals myelatrophy, intervertebral disc bulge at C5-C7 and L1-L3. ate shape and size, they were reassembled and immobilized on the dorsal surface of the anti-syphilis therapy with 4.8 million units of proximal tibia with two 4.5 mm cannulated penicillin intravenously every 12 hours for 14 screws (Figure 7D). days on the basis of established etiology prior to being accepted for surgery. Sensory recovery The reconstructed tibial plateau demonstrated of the bilateral lower limbs occurred two weeks sufficient surface contact with the femoral si- after treatment. A retest of the patient’ RPR de. Finally, a bend-forming 13-hole limited con- was weakly positive (+), which indirectly proved tact-locking compression plate (LC-LCP) was that this therapy was effective. applied medially and secured with locking screws in addition to conventional bicortical sc- The procedure was performed with the patient rews. An additional 14-hole locking compres- supine with a tourniquet around his thigh inflat- sion plate (LCP) was fastened anteriorly and

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Discussion

The surgical therapeutic management for Charcot knee arthropathy remains controver- sial. Traditionally, TKA was discouraged as a management strategy of Charcot arthropathy due to the risk of secondary osteoporosis and a high incidence rate of prosthetic loosening. Recently there have been several reports of TKA being used in Charcot knee arthropathy, yet the curative effect is still ambiguous, especially the long-term prognosis. A review of the available literature revealed that the total complication rate is approximately 50% (35 complications on 72 knees), including dislocation (7%), periprosthetic fractures (7%), deep venous thrombosis (DVT, 8%), disruption of the quadriceps (4%), deep infection (3%) and other complications [8]. Kim et al. reported that 19 knees diagnosed with Charcot arthropathy accepted TKA with 47% of the total cases result- ing in various complications, and only 53% of the patients with severe Charcot arthropathy showing satisfactory results at a 5-year follow- Figure 6. Emission computed tomography performed 2.5 years previously showed nuclide concentration up [9]. Arthrodesis is commonly the recom- of right hip and left knee. mended choice of surgical treatment for Cha- rcot arthropathy, especially in cases with severe instability, soft-tissue laxity and bony destruc- secured with bicortical screws (Figure 7E). It tion [10]. was necessary to curve the plates, so that they would conform to the shape of the tibia and It is recognized that knee fusion is not easily femur. Postoperative limb length measurement achieved in Charcot arthropathy because of showed that the left leg was 1.5 cm shorter poor bone quality and underlying fundamental than the contralateral side, which was within diseases. Furthermore, for cases with large an acceptable range. An above-the-knee plas- bone defects, the situation appears more in- ter support was also fitted for 2 months post- tractable. To our knowledge, although there operatively before weight-bearing training. were reports of using fibulae or orthobiologics for bone grafts, no previous reports of the sur- Two months after surgery there was radiologi- gical treatment of Charcot knee arthropathy cal evidence of callus formation so the patient with patella grafts have been published. Ac- commenced partial weight-bearing with crutch hieving long-term stability in a patient with ambulation (Figure 8A). Five months postoper- Charcot arthropathy was considered extremely atively radiographs confirmed gross knee fu- important, especially for cases with severe sion and he was allowed to fully weight bear. bone loss. It was necessary that a difficult sec- The final outcome obtained 10 months after ondary revision procedure was avoided, consid- surgery showed excellent consolidation without ering the habitual dislocation of the right deformity and with well-maintained lower limb Charcot hip and the underlying possibility of alignment (Figure 8B). His gait was basically developing Charcot arthropathy in the contra- steady without crutches with no complaints of lateral hip or knee, highlighting the importance pain and with no collection of joint fluid Figure( of maintaining limb length and bone stock. This 8C). case was characterized by a large bone defi- ciency at the posterior of the tibial plateau, Informed consent was obtained from the pa- which decreased the contact area of the proxi- tient for publication of this manuscript using mal tibia and distal femur, further reducing the his information and images. probability of fusion. Given the difficulty of this

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Figure 7. A. A Mid-longitudinal incision bypassing the patella of left knee; B. Large bone defects of posterior tibial plateau were observed during the operation; C. The intact patella was sawed into two half of pieces; D. The tibial plateau was structurally reconstructed with reassembled patella graft and immobilized with two cannulated screws; E. Two bend-forming locking compression plates were placed anteriorly and medially of the knee joint. case, it was important to secure a bone graft of rebuild the tibial plateau. This technique has sufficient volume to provide good mechanical several theoretical advantages. Above all, this stability. The technique described above ov- method clearly created conditions that were ercame the challenges by creating a larger sur- more physiological inside the main pathologic face area for bony union and rigid stability with lesion compared with other orthobiologics. dual locking compression plates. Autogenous bone is regarded as the reference standard for bone graft material, because it Vascularized fibular grafts have been the most provides the specific elements required to gen- commonly-used autogenous bone for recon- erate and maintain bone, including scaffolding struction of skeletal defects in knee arthrode- for osteoconduction, growth factors for osteo- sis. Several reports have also suggested utiliz- induction and progenitor cells for osteogenesis ing of other orthobiologics. Voss introduced the [13]. Secondly, the choice of the patella as a concept of artificial arthrodesis by use of a source of the autogenous bone resulted in a cement spacer as an alternative in treating better fit of the graft and host bone than occurs cases with severe bone loss [11]. Capanna et with a fibula or other orthobiologic because of al. used modular nails with metal, polyethylene the geometry of the bone surface. In addition, or cement with gentamycin spacers around the using the patella as a bone graft avoided creat- central coupler with gentamycin for patients ing an additional incision on the shank, certain- with bony defects [12]. Both methods proved ly decreasing the risk of postoperative infection feasible. In this case, the patella was used and shortening recovery time compared to har- innovatively as an autologous bone graft to vesting the fibula. Although no published data

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insufficient angulation may increase the limb-length dis- crepancy, instability of gait and finally leading to the fail- ure of fusion. The fixation methods in knee arthrodesis include external fixator, intramedullary nail and plate fixation. Although intramedullary nailing produces the highest rate of fusion of the three methods above, considering the loss of bone, inferior bone stock and mild anemia in this present case, which would contraindicate of external fixation and intramedullary nailing, the double-plating fixation method was chosen. Kuo et al. reported using a dual-LCP technique to achieve fusion in 3 patients with a 100% suc- cess rate [15]. This technique has the advantage of providing axial compression of the Figure 8. A. Plain X-ray image obtained 2 months postoperatively shows ini- fusion construct and fixed tiation of union process; B. Anteroposterior and lateral radiography of left angle stability with the lock- knee 10 months after knee arthrodesis exhibited plenty formation of osteo- ing screws, which cannot be tylus and favourable fusion condition; C. Steady standing posture 10 months after surgery without any upholder. achieved through intramedullary nailing. The use of an above-the-knee cast for an Table 2. Pre-operation and post-operation SF-36 score average of 18 weeks (ranging SF-36 PF RP BP GH VT SF RE MH PCS MCS from 12 to 53 weeks) postoperatively prior to weight bear- Pre-operation 20 0 40 30 70 33 67 60 90 230 ing is also recommended in Post-operation 40 50 74 47 75 89 100 64 211 328 order to restrict body activity Note: PF, Physical functioning; RP, Role-physical; BP, Bodily pain; GH, General and promote fusion [16]. health; VT, Vitality; SF, Social functioning; RE, Role-emotional; MH, Mental health; PCS, Physical component summary; MCS, Mental component summary. The At a follow-up visit 10 months patient’s SF-36 score increased markedly compared to preoperative status, which indicating his quality of life had improved a lot both physically and mentally after postoperatively, the patient receiving the surgery. showed no complications around the wound and neither instability nor pain were pre- has compared the clinical results of a patella sented. He could walk on his own for 500 graft technique and other orthobiologics for meters without a cane or walker. His SF-36 Charcot knee fusion, our data suggest that score had greatly improved compared to before patella autograft may accelerate bone union, the surgery (Table 2). A recent plain radiograph minimize hardware-related problems, and showed that a large quantity of osteotylus had result in a lower risk of infection. formed and the joint was basically fused with no sign of internal fixation loosening or break- The prerequisites for successful fusion include ing. In this case, the patient’s gait was steady full contact of cancellous bone surfaces, satis- and the quality of his daily life had significantly factory limb alignment and rigid fixation. The improved compared with preoperatively. Alth- ideal limb alignment for knee fusion should be ough there is currently no apparent complica- in the “neutral range”: 5° to 7° of valgus and tion, long-term follow-up is necessary and im- 10° to 20° of flexion [14]. Either excessive or portant.

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In conclusion, a satisfactory outcome was ob- [5] Gupta R. A short history of neuropathic ar- tained without infection or nonunion in per- thropathy. Clin Orthop Relat Res 1993; 296: forming a successful knee arthrodesis for a 43-49. patient suffering from Charcot arthropathy, wi- [6] Zeng M, Xie J and Hu Y. Total knee arthroplasty th the use of a free, reassembled patella graft. in patients with Charcot joints. Knee Surg Sports Traumatol Arthrosc 2016; 24: 1-6. Based on our understanding of the literature [7] Fullerton BD and Browngoehl LA. Total knee and our experience with this case, application arthroplasty in a patient with bilateral Charcot of patella graft should be considered in similar knees. Arch Phys Med Rehabil 1997; 78: 780- cases with severe bone defect when perform- 782. ing knee arthrodesis. [8] Babazadeh S, Stoney JD, Lim K and Choong PF. Arthroplasty of a Charcot knee. Orthop Rev Acknowledgements (Pavia) 2010; 2: e17. [9] Kim YH, Kim JS and Oh SW. Total knee arthro- The authors are grateful to Jessica He (Uni- plasty in neuropathic arthropathy. J Bone Joint versity of Waterloo, Canada) for carefully review- Surg Br 2002; 84: 216-219. ing and editing the manuscript. [10] Vince KG, Cameron HU, Hungerford DS, Laskin RS, Ranawat CS and Scuderi GR. What would Disclosure of conflict of interest you do?: case challenges in knee surgery. J Arthroplasty 2005; 20: 44-50. None. [11] Voss FR. A new technique of limb salvage after infected revision total knee arthroplasty-the Address correspondence to: Ming Xu, Department journal of arthroplasty. J Arthroplasty 2001; of Orthopaedic Surgery, The First Affiliated Hospital 16: 524-528. of Soochow University, No. 188 Shizi Street, Suzhou [12] Capanna R, Biagini R, Ruggieri P, Bettelli G, 215000, Jiangsu Province, P. R. China. E-mail: min- Casadei R and Campanacci M. Temporary re- section-arthrodesis of the knee using an intra- [email protected] medullary rod and bone cement. Int Orthop References 1989; 13: 253. [13] Vaccaro AR. The role of the osteoconductive [1] Womack J. Charcot arthropathy versus osteo- scaffold in synthetic bone graft. Orthopedics myelitis: evaluation and management. Orthop 2002; 25: s571. Clin North Am 2017; 48: 241-247. [14] Conway JD, Mont MA and Bezwada HP. [2] Sudanese A, Toni A, Moscato M, Ciaroni D, Arthrodesis of the knee. J Bone Joint Surg Am Marraro D and Giunti A. Radiographic and ther- 2004; 86-A: 835. apeutic aspects of neurogenic arthropathy. [15] Kuo AC, Meehan JP and Lee M. Knee fusion Chir Organi Mov 1992; 77: 325. using dual platings with the locking compres- [3] Viens NA, Watters TS, Vinson EN and Brigman sion plate. J Arthroplasty 2005; 20: 772-776. BE. Case report: neuropathic arthropathy of [16] Nichols SJ, Landon GC and Tullos HS. the hip as a sequela of undiagnosed tertiary Arthrodesis with dual plates after failed total syphilis. Clin Orthop Relat Res 2010; 468: knee arthroplasty. J Bone Joint Surg Am 1991; 3126. 73: 1020-1024. [4] Zhang YQ, Huang M, Jia XY, Zou YF and Chen D. A clinical study of new cases of parenchymal neurosyphilis: has tabes dorsalis disappeared or been missed? J Neuropsychiatry Clin Neurosci 2015; 27: e17-21.

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